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Is treatment with hydroxychloroquine effective in surfactant protein C deficiency?

Abstract
We present the case of two twin brothers with surfactant protein C deficiency who were treated with hydroxychloroquine for three years, with apparent success. The exact physiopathology of this disease is not known and there is no specific treatment for it. There is merely news from a few previous descriptions in the literature about the use of hydroxychloroquine for surfactant protein C deficiency with satisfactory results. Two years after the treatment was withdrawn, the twins were evaluated once again: they presented no new infections, growth and general state were normal and chest CT showed a notable additional reduction in the interstitial pneumopathy. These data seem to cast some doubt on the efficacy of hydroxychloroquine, and they suggest that the clinical improvement was simply the natural evolution of the disease.
AuthorsIngrid Rabach, Furio Poli, Floriana Zennaro, Claudio Germani, Alessandro Ventura, Egidio Barbi
JournalArchivos de bronconeumologia (Arch Bronconeumol) Vol. 49 Issue 5 Pg. 213-5 (May 2013) ISSN: 1579-2129 [Electronic] Spain
PMID23137777 (Publication Type: Case Reports, Journal Article)
CopyrightCopyright © 2012 SEPAR. Published by Elsevier Espana. All rights reserved.
Chemical References
  • Pulmonary Surfactant-Associated Protein C
  • Hydroxychloroquine
Topics
  • Disease Progression
  • Diseases in Twins (drug therapy, genetics)
  • Dyspnea (etiology)
  • Failure to Thrive (etiology)
  • Humans
  • Hydroxychloroquine (therapeutic use)
  • Infant, Newborn
  • Male
  • Pulmonary Alveolar Proteinosis (complications, diagnostic imaging, drug therapy, genetics)
  • Pulmonary Surfactant-Associated Protein C (deficiency)
  • Respiratory Insufficiency (etiology)
  • Tomography, X-Ray Computed
  • Twins, Monozygotic

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